Document Detail


Electrophysiologic evidence for anterior horn cell disease in amyoplasia.
MedLine Citation:
PMID:  20610128     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Amyoplasia is the most common subtype of arthrogryposis multiplex congenita. Children exhibit congenital muscle aplasia or hypoplasia, resulting in symmetric limb contractures that occur in a characteristic pattern. A high incidence of midforehead capillary hemangiomas, micrognathia, and minor genital anomalies also occurs with this disorder. The etiology and specific site of injury in amyoplasia remain unclear. Previous muscle biopsy and electrophysiologic studies could not differentiate between neurogenic or myogenic causes. We describe five children with amyoplasia. Four children demonstrated electrophysiologic features consistent with motor neuronopathy. A careful segmental needle electromyography study revealed variable involvement at different myotomes. We hypothesize that the anterior horn cell may represent the site of disease in a subset of children with amyoplasia.
Authors:
John N Gaitanis; Hugh J McMillan; Allan Wu; Basil T Darras
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatric neurology     Volume:  43     ISSN:  1873-5150     ISO Abbreviation:  Pediatr. Neurol.     Publication Date:  2010 Aug 
Date Detail:
Created Date:  2010-07-08     Completed Date:  2011-01-14     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8508183     Medline TA:  Pediatr Neurol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  142-7     Citation Subset:  IM    
Copyright Information:
Copyright 2010 Elsevier Inc. All rights reserved.
Affiliation:
Department of Neurology, Hasbro Children's Hospital, Brown Medical School, Providence, Rhode Island, USA.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Arthrogryposis / complications*,  diagnosis*
Electromyography / methods*
Female
Humans
Infant
Infant, Newborn
Male
Motor Neuron Disease / complications*,  diagnosis*
Muscle, Skeletal / physiopathology
Neural Conduction / physiology
Neurologic Examination

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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